Esen Alparslan, Dolanmaz Doğan, Kalayci Abdullah, Günhan Omer, Avunduk Mustafa Cihat
Department of Oral and Maxillofacial Surgery, Medical Park Hospital, Antalya, Turkey.
Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2010 Feb;109(2):e53-8. doi: 10.1016/j.tripleo.2009.10.015.
Localized Langerhans cell histiocytosis (LLCH), formerly known as eosinophilic granuloma, mainly affects the skull, mandible, vertebrae, and ribs in children and the long bones of adults. Symptoms range from none to pain, swelling, and tenderness over the site of the lesion. General malaise and fever occasionally are present. Radiographically, lesions appear as radiolucent areas with well demarcated borders. LLCH may resolve spontaneously after biopsy in a period of months to years. However, if features include continuous pain, decrease of function, pathologic fractures, migration and resorption of teeth, or rapid progression, then active treatment needs to be considered. Treatment approaches include surgery, radiotherapy, chemotherapy, and intralesional injection of corticosteroids. In children with mandibular LLCH, 1 dose of methyprednisolone succinate injection has proven to be adequate. However, injections have not been performed in cases involving pathologic fracture. We report a new case of LLCH of the mandible that caused a pathologic fracture in an adult patient. Repeated intralesional corticosteroid injections resulted in fracture line disappearance within 14 months and lesion healing by the end of the 36-month follow-up.
局限性朗格汉斯细胞组织细胞增多症(LLCH),以前称为嗜酸性肉芽肿,主要影响儿童的颅骨、下颌骨、椎骨和肋骨以及成人的长骨。症状从无到病变部位疼痛、肿胀和压痛不等。偶尔会出现全身不适和发热。在影像学上,病变表现为边界清晰的透光区。LLCH在活检后的数月至数年期间可能会自行消退。然而,如果出现持续疼痛、功能减退、病理性骨折、牙齿移位和吸收或快速进展等特征,则需要考虑积极治疗。治疗方法包括手术、放疗、化疗和病灶内注射皮质类固醇。在下颌骨LLCH患儿中,已证明1剂琥珀酸甲泼尼龙注射液就足够了。然而,在涉及病理性骨折的病例中尚未进行注射治疗。我们报告了1例成年患者下颌骨LLCH导致病理性骨折的新病例。反复病灶内注射皮质类固醇导致骨折线在14个月内消失,在36个月随访结束时病变愈合。